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131I-间碘苄胍治疗复发或难治性神经母细胞瘤后的复发和进展的解剖学模式。

Anatomic patterns of relapse and progression following treatment with I-MIBG in relapsed or refractory neuroblastoma.

作者信息

Fishel Ben Kenan Rotem, Polishchuk Alexei L, Hawkins Randall A, Braunstein Steve E, Matthay Katherine K, DuBois Steven G, Haas-Kogan Daphne A

机构信息

Department of Pediatrics, Phoenix Children's Hospital, Phoenix, Arizona, USA.

Bellingham Radiation Oncology, PLLC, Bellingham, Washington, USA.

出版信息

Pediatr Blood Cancer. 2022 Feb;69(2):e29396. doi: 10.1002/pbc.29396. Epub 2021 Oct 18.

Abstract

OBJECTIVES

Patients with metaiodobenzylguanidine (MIBG)-avid relapsed or refractory neuroblastoma after initial therapy may exhibit transient responses to salvage treatment with iodine-131 metaiodobenzylguanidine ( I-MIBG). It is unclear whether disease progression following I-MIBG treatment occurs in previously involved versus new anatomic sites of disease. Understanding this pattern of relapse will inform the use of consolidation therapy following I-MIBG administration.

METHODS

Patients with relapsed or refractory metastatic MIBG-avid neuroblastoma or ganglioneuroblastoma, who received single-agent I-MIBG, had stable or responding disease 6-8 weeks following I-MIBG, but subsequently experienced disease progression were included. MIBG scans were reviewed to establish anatomic and temporal evolution of MIBG-avid disease.

RESULTS

A total of 84 MIBG-avid metastatic sites were identified immediately prior to MIBG therapy in a cohort of 12 patients. At first progression, a total of 101 MIBG-avid sites were identified, of which 69 (68%) overlapped with pre-treatment disease sites, while 32 (32%) represented anatomically new disease areas. Eight of 12 patients had one or more new MIBG-avid sites at first progression. Of the 69 involved sites at progression that overlapped with pre-treatment disease, 11 represented relapsed sites that had cleared following MIBG therapy, two were persistent but increasingly MIBG-avid, and 56 were stably persistent.

CONCLUSIONS

Previously involved anatomic disease sites predominate at disease progression following I-MIBG treatment. Nevertheless, the majority of patients progressed in at least one new anatomic disease site. This suggests that consolidation focal therapies targeting residual disease sites may be of limited benefit in preventing systemic disease progression following I-MIBG treatment of relapsed or refractory neuroblastoma.

摘要

目的

接受初始治疗后对间碘苄胍(MIBG)摄取阳性的复发性或难治性神经母细胞瘤患者,可能对用碘-131间碘苄胍(I-MIBG)进行挽救治疗表现出短暂反应。尚不清楚I-MIBG治疗后疾病进展是发生在先前受累的疾病解剖部位还是新的解剖部位。了解这种复发模式将为I-MIBG给药后巩固治疗的应用提供依据。

方法

纳入复发性或难治性转移性MIBG摄取阳性的神经母细胞瘤或神经节神经母细胞瘤患者,这些患者接受单药I-MIBG治疗,在I-MIBG治疗后6-8周疾病稳定或有反应,但随后病情进展。回顾MIBG扫描结果以确定MIBG摄取阳性疾病的解剖和时间演变。

结果

在一组12例患者中,在MIBG治疗前共确定了84个MIBG摄取阳性的转移部位。在首次病情进展时,共确定了101个MIBG摄取阳性部位,其中69个(68%)与治疗前疾病部位重叠,而32个(32%)代表解剖学上新的疾病区域。12例患者中有8例在首次病情进展时有一个或多个新的MIBG摄取阳性部位。在病情进展时与治疗前疾病重叠的69个受累部位中,11个代表MIBG治疗后已清除的复发部位,2个为持续存在但MIBG摄取增加的部位,56个为稳定持续存在的部位。

结论

I-MIBG治疗后疾病进展时,先前受累的解剖疾病部位占主导。然而,大多数患者至少在一个新的解剖疾病部位出现病情进展。这表明,针对残留疾病部位的巩固性局部治疗在预防复发性或难治性神经母细胞瘤I-MIBG治疗后全身性疾病进展方面可能益处有限。

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